目的:自身免疫的发作和恶化,在Cushing综合征缓解后,已经报道了炎症或类固醇反应性疾病,导致在缓解后区分新情况和预期症状方面面临挑战。我们描述了一例42岁男子在库欣综合征手术治愈12个月后被诊断为新发病结节病的病例,并综合了有关库欣综合征缓解后出现的新发病情况的现有文献报道。
方法:在Medline进行了范围审查,Epub,Ovid和PubMed.包括病例报告和病例系列,详细介绍了库欣综合征缓解后出现新发疾病的成年患者。
结果:总计,筛选了1641篇文章,对138项全文研究进行了资格评估,并纳入了43项研究,其中84例(包括我们的病例)被确认。大多数患者为女性(85.7%),报告年龄中位数为39.5岁(IQR=13).甲状腺疾病是最常见的疾病(48.8%),其次是结节病(15.5%)。牛皮癣,淋巴细胞性垂体炎,特发性颅内高压,多发性硬化症,类风湿性关节炎,报告狼疮和血清阴性关节炎超过一例。库欣缓解和从头诊断之间的中位持续时间为4.1个月(IQR=3.75)。这些病人中,59.5%在发病时接受皮质类固醇治疗。
结论:我们的范围审查发现了几例库欣综合征缓解后出现的从头疾病。它们主要发生在女性中,在缓解后的一年内。临床医生应该意识到新的症状,特别是在治疗库欣综合征后的第一年,除肾上腺功能不全或糖皮质激素戒断综合征外,可能是多种疾病的表现。
OBJECTIVE: Onset and exacerbation of autoimmune, inflammatory or steroid-responsive conditions have been reported following the remission of Cushing syndrome, leading to challenges in distinguishing a new condition versus expected symptomatology following remission. We describe a
case of a 42-year-old man presenting with new-onset sarcoidosis diagnosed 12 months following the surgical cure of Cushing syndrome and synthesise existing literature reporting on de novo conditions presenting after Cushing syndrome remission.
METHODS: A scoping review was conducted in Medline, Epub, Ovid and PubMed.
Case reports and
case series detailing adult patients presenting with new-onset conditions following Cushing syndrome remission were included.
RESULTS: In total, 1641 articles were screened, 138 full-text studies were assessed for eligibility, and 43 studies were included, of which 84 cases (including our
case) were identified. Most patients were female (85.7%), and the median reported age was 39.5 years old (IQR = 13). Thyroid diseases were the most commonly reported conditions (48.8%), followed by sarcoidosis (15.5%). Psoriasis, lymphocytic hypophysitis, idiopathic intracranial hypertension, multiple sclerosis, rheumatoid arthritis, lupus and seronegative arthritis were reported in more than one
case. The median duration between Cushing remission and de novo condition diagnosis was 4.1 months (IQR = 3.75). Of those patients, 59.5% were receiving corticosteroid therapy at the time of onset.
CONCLUSIONS: Our scoping review identified several cases of de novo conditions emerging following the remission of Cushing syndrome. They occurred mostly in women and within the year following remission. Clinicians should remain aware that new symptoms, particularly in the first year following the treatment of Cushing syndrome, may be manifestations of a wide range of conditions aside from adrenal insufficiency or glucocorticoid withdrawal syndrome.